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http://purl.uniprot.org/citations/31582721http://www.w3.org/1999/02/22-rdf-syntax-ns#typehttp://purl.uniprot.org/core/Journal_Citation
http://purl.uniprot.org/citations/31582721http://www.w3.org/2000/01/rdf-schema#comment"LRRC7 has been identified as a candidate gene for severe childhood emotional dysregulation. Direct experimental evidence for a role of LRRC7 in the disease is needed, as is a better understanding of its impact on neuronal structure and signaling, and hence potential treatment targets. Here, we generated and analyzed an Lrrc7 mutant mouse line. Consistent with a critical role of LRRC7 in emotional regulation, mutant mice had inappropriate juvenile aggressive behavior and significant anxiety-like behavior and social dysfunction in adulthood. The pivotal role of mGluR5 signaling was demonstrated by rescue of behavioral defects with augmentation of mGluR5 receptor activity by 3-Cyano-N-(1,3-diphenyl-1H-pyrazol-5-yl)benzamide (CDPPB). Intra-peritoneal injection of CDPPB alleviated abnormal juvenile behavior, as well as anxiety-like behavior and hypersociability at adulthood. Furthermore, mutant primary neurons had impaired neurite outgrowth which was rescued by CDPPB treatment. In conclusion, Lrrc7 mutant mice provide a valuable tool to model childhood emotional dysregulation and persistent mental health comorbidities. Moreover, our data highlight an important role of LRRC7 in mGluR5 signaling, which is a potential new treatment target for anxiety and social dysfunction."xsd:string
http://purl.uniprot.org/citations/31582721http://purl.org/dc/terms/identifier"doi:10.1038/s41398-019-0580-9"xsd:string
http://purl.uniprot.org/citations/31582721http://purl.uniprot.org/core/author"Li Q."xsd:string
http://purl.uniprot.org/citations/31582721http://purl.uniprot.org/core/author"Chan S.Y."xsd:string
http://purl.uniprot.org/citations/31582721http://purl.uniprot.org/core/author"Chan A.K.W."xsd:string
http://purl.uniprot.org/citations/31582721http://purl.uniprot.org/core/author"McAlonan G."xsd:string
http://purl.uniprot.org/citations/31582721http://purl.uniprot.org/core/author"Chong C.H."xsd:string
http://purl.uniprot.org/citations/31582721http://purl.uniprot.org/core/author"Mak P.H.S."xsd:string
http://purl.uniprot.org/citations/31582721http://purl.uniprot.org/core/author"Leung E.H.W."xsd:string
http://purl.uniprot.org/citations/31582721http://purl.uniprot.org/core/author"Ng C.C.P."xsd:string
http://purl.uniprot.org/citations/31582721http://purl.uniprot.org/core/author"Tan V.H."xsd:string
http://purl.uniprot.org/citations/31582721http://purl.uniprot.org/core/date"2019"xsd:gYear
http://purl.uniprot.org/citations/31582721http://purl.uniprot.org/core/name"Transl Psychiatry"xsd:string
http://purl.uniprot.org/citations/31582721http://purl.uniprot.org/core/pages"244"xsd:string
http://purl.uniprot.org/citations/31582721http://purl.uniprot.org/core/title"Lrrc7 mutant mice model developmental emotional dysregulation that can be alleviated by mGluR5 allosteric modulation."xsd:string
http://purl.uniprot.org/citations/31582721http://purl.uniprot.org/core/volume"9"xsd:string
http://purl.uniprot.org/citations/31582721http://www.w3.org/2004/02/skos/core#exactMatchhttp://purl.uniprot.org/pubmed/31582721
http://purl.uniprot.org/citations/31582721http://xmlns.com/foaf/0.1/primaryTopicOfhttps://pubmed.ncbi.nlm.nih.gov/31582721
http://purl.uniprot.org/uniprot/#_A0A0G2JDT9-mappedCitation-31582721http://www.w3.org/1999/02/22-rdf-syntax-ns#objecthttp://purl.uniprot.org/citations/31582721
http://purl.uniprot.org/uniprot/#_A0A0G2JFZ5-mappedCitation-31582721http://www.w3.org/1999/02/22-rdf-syntax-ns#objecthttp://purl.uniprot.org/citations/31582721
http://purl.uniprot.org/uniprot/#_E9Q6L9-mappedCitation-31582721http://www.w3.org/1999/02/22-rdf-syntax-ns#objecthttp://purl.uniprot.org/citations/31582721
http://purl.uniprot.org/uniprot/#_A0A571BEA5-mappedCitation-31582721http://www.w3.org/1999/02/22-rdf-syntax-ns#objecthttp://purl.uniprot.org/citations/31582721
http://purl.uniprot.org/uniprot/#_B2BH30-mappedCitation-31582721http://www.w3.org/1999/02/22-rdf-syntax-ns#objecthttp://purl.uniprot.org/citations/31582721
http://purl.uniprot.org/uniprot/#_E9QMC2-mappedCitation-31582721http://www.w3.org/1999/02/22-rdf-syntax-ns#objecthttp://purl.uniprot.org/citations/31582721